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Presacral malakoplakia presenting as foot drop: a case report

BACKGROUND: Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to develop in association with chronic or recurrent infections, typically of the urinary tract. A specific defect in innate...

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Autores principales: Yates, Tom A., Devlin, Katie, Arnaout, Abed, Hurt, William, Stone, Neil, Everett, Kate V., Pittman, Alan, Patel, Hardik, Heenan, Susan, Hart, Paul, Harrison, Thomas S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10080903/
https://www.ncbi.nlm.nih.gov/pubmed/37024963
http://dx.doi.org/10.1186/s13256-023-03883-4
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author Yates, Tom A.
Devlin, Katie
Arnaout, Abed
Hurt, William
Stone, Neil
Everett, Kate V.
Pittman, Alan
Patel, Hardik
Heenan, Susan
Hart, Paul
Harrison, Thomas S.
author_facet Yates, Tom A.
Devlin, Katie
Arnaout, Abed
Hurt, William
Stone, Neil
Everett, Kate V.
Pittman, Alan
Patel, Hardik
Heenan, Susan
Hart, Paul
Harrison, Thomas S.
author_sort Yates, Tom A.
collection PubMed
description BACKGROUND: Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to develop in association with chronic or recurrent infections, typically of the urinary tract. A specific defect in innate immunity has been described. In the absence of randomized controlled trials, management is based on an understanding of the biology and on case reports. CASE PRESENTATION: Here we describe a case of presacral malakoplakia in a British Indian woman in her late 30s, presenting with complex unilateral foot drop. Four years earlier, she had suffered a protracted episode of intrapelvic sepsis following a caesarean delivery. Resection of her presacral soft tissue mass was not possible. She received empiric antibiotics, a cholinergic agonist, and ascorbic acid. She responded well to medical management both when first treated and following a recurrence of symptoms after completing an initial 8 months of therapy. Whole exome sequencing of the patient and her parents was undertaken but no clear causal variant was identified. CONCLUSIONS: Malakoplakia is uncommon but the diagnosis should be considered where soft tissue masses develop at the site of chronic or recurrent infections. Obtaining tissue for histological examination is key to making the diagnosis. This case suggests that surgical resection is not always needed to achieve a good clinical and radiological outcome. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s13256-023-03883-4.
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spelling pubmed-100809032023-04-08 Presacral malakoplakia presenting as foot drop: a case report Yates, Tom A. Devlin, Katie Arnaout, Abed Hurt, William Stone, Neil Everett, Kate V. Pittman, Alan Patel, Hardik Heenan, Susan Hart, Paul Harrison, Thomas S. J Med Case Rep Case Report BACKGROUND: Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to develop in association with chronic or recurrent infections, typically of the urinary tract. A specific defect in innate immunity has been described. In the absence of randomized controlled trials, management is based on an understanding of the biology and on case reports. CASE PRESENTATION: Here we describe a case of presacral malakoplakia in a British Indian woman in her late 30s, presenting with complex unilateral foot drop. Four years earlier, she had suffered a protracted episode of intrapelvic sepsis following a caesarean delivery. Resection of her presacral soft tissue mass was not possible. She received empiric antibiotics, a cholinergic agonist, and ascorbic acid. She responded well to medical management both when first treated and following a recurrence of symptoms after completing an initial 8 months of therapy. Whole exome sequencing of the patient and her parents was undertaken but no clear causal variant was identified. CONCLUSIONS: Malakoplakia is uncommon but the diagnosis should be considered where soft tissue masses develop at the site of chronic or recurrent infections. Obtaining tissue for histological examination is key to making the diagnosis. This case suggests that surgical resection is not always needed to achieve a good clinical and radiological outcome. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s13256-023-03883-4. BioMed Central 2023-04-07 /pmc/articles/PMC10080903/ /pubmed/37024963 http://dx.doi.org/10.1186/s13256-023-03883-4 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Yates, Tom A.
Devlin, Katie
Arnaout, Abed
Hurt, William
Stone, Neil
Everett, Kate V.
Pittman, Alan
Patel, Hardik
Heenan, Susan
Hart, Paul
Harrison, Thomas S.
Presacral malakoplakia presenting as foot drop: a case report
title Presacral malakoplakia presenting as foot drop: a case report
title_full Presacral malakoplakia presenting as foot drop: a case report
title_fullStr Presacral malakoplakia presenting as foot drop: a case report
title_full_unstemmed Presacral malakoplakia presenting as foot drop: a case report
title_short Presacral malakoplakia presenting as foot drop: a case report
title_sort presacral malakoplakia presenting as foot drop: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10080903/
https://www.ncbi.nlm.nih.gov/pubmed/37024963
http://dx.doi.org/10.1186/s13256-023-03883-4
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